Uncommon situation and presentation of chronic sclerosing sialadenitis

Adouly, T.; Adnane, C.; Housni, Y.; Rouadi, S.; Abada, R.; Roubal, M.; Mahtar, M.

doi:10.1016/j.ijporl.2016.01.020

Cited by 6 publications

(7 citation statements)

References 7 publications

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“…A significant proportion of cases previously diagnosed as Miculicz's disease (dacryoadenitis and enlargement of parotid and submandibular glands) and Kuttner tumor (chronic sclerosing sialadenitis of submandibular gland) are now considered IgG4-RD. IgG4-related chronic sclerosing sialadenitis is a rare event in children [11,12]. We report here a case of a unexpected diagnosis of IgG4-related sialadenitis in a boy previously diagnosed as affected by juvenile recurrent parotitis.…”

Section: Introductionmentioning

confidence: 86%

Chronic sclerosing sialadenitis IgG4-related in a boy with recurrent parotitis: a case report

Timeus¹,

Calvo²,

Caci³

et al. 2021

Preprint

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BackgroundIgG4-related diseases are a group of immune-mediated diseases characterized by a lymphoplasmacytic infiltrate with a prevalence of IgG4-positive plasma cells, storiform fibrosis, obliterative phlebitis. Autoimmune pancreatitis, sialadenitis, dacryoadenitis and retroperitoneal fibrosis are the most frequent manifestations. IgG4-related sialadenitis usually affects submandibular glands and is very rare in children. We report here a case of an unexpected diagnosis of IgG4-related sialoadenitis in a boy previously diagnosed as affected by juvenile recurrent parotitis.Case presentationA six-year old boy presented to our centre with left parotid swelling about 4x3 cm, tender, soft in consistency, with the overlying skin red and warm. He had low-grade fever and a mild enlargement of left cervical lymph nodes. In the last two years he had five episodes of parotitis and a diagnosis of juvenile recurrent parotitis.. The general conditions were good, and the remainder of the physical examination was normal. At the ultrasonography left parotid was enlarged, inhomogeneous, with a colliquated intraparotid lymph node and no evidence of sialolithiasis. Laboratory tests were normal, except for an increase of white blood cells and positivity for IgM and IgG anti-VCA, with anti-EBNA e anti-EA I negative. The patient was treated with cephalosporins and azitromycin, but after 10 days the parotid became fluctuating and the patient underwent to drainage and biopsy of the gland. He was discharged after 3 days of observation, without any complications, and treated with a further course of cefpodoxime and betametasone, with a good clinical response.Investigations for aerobic and anaerobic bacteria, mycobacteria and Bartonella on the pus were negative. Histology showed marked fibrosis and histiocytic and lymphoplasmacellular infiltration. The plasma cells were polyclonal and mostly expressed IgG4 subclass immunoglobulins. The final diagnosis was chronic sclerosing sialadenitis IgG4-related in recurrent parotitis and recent EBV infection. ConclusionsThe interest of this case comes from the rarity of IgG4-related sialadenitis in children. It is not possible to argue with certainty that the previous episodes of parotitis were manifestations of a IgG4-related disease, due to the lack of histological data. Serum IgG4 in our patient were normal, as described in literature in 30% of cases.

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Section: Introductionmentioning

confidence: 86%

Chronic sclerosing sialadenitis IgG4-related in a boy with recurrent parotitis: a case report

Timeus¹,

Calvo²,

Caci³

et al. 2021

Preprint

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“…Although there have been reports of unilateral parotid gland involvement of CSS, its presentation is rare, especially in pediatric African-American patients [10][11][12]. Due to the relatively novel status of CSS within the umbrella of IgG4-RD, there is no meaningful epidemiologic data.…”

Section: Presentationmentioning

confidence: 99%

Pediatric Parotid Chronic Sclerosing Sialadenitis in an African-American Female: A Rare Case and Review of the Literature

Keidar

Shermetaro

Kwartowitz³

2020

Cureus

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Chronic sclerosing sialadenitis (CSS) or Küttner tumor is an under-recognized, benign fibroinflammatory disease most commonly seen in the submandibular gland of older adult males. Sialolithiasis or bacterial infection was first suspected as an etiology, but CSS is now considered an immunoglobulin G4-related disease (IgG4-RD). IgG4-RD can affect almost every organ in the body, characterized by organ fibrosis with IgG4-positive plasmacytes. Numerous autoimmune-related diseases have been unified under IgG4-RD, including Mikulicz disease (MD), autoimmune pancreatitis, Reidel’s thyroiditis, and others. In any organ, IgG4-RD can present similar to malignancy. Due to the ability to mimic malignancy, it is crucial to be aware of this under-recognized clinical entity. CSS is currently of broad and high clinical interest due to increased understanding, multiorgan involvement, and more clearly defined criteria. To increase awareness of this disease, we describe a rare presentation of CSS with a literature review.

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“…This is believed to be due to the secretions within the submandibular gland being more mucoid and less serous than that of the parotid glands, making stone formation more likely. 5,8 A second group of patients are those affected by systemic fibrosis where it may be that multiple glands may be susceptible. This entity of chronic sclerosing sialadenitis and associated IgG4-positive plasma cell infiltration can be found in patients with Sjögren syndrome, chronic sclerosing inflammatory fibrosis, and sialadenitis.…”

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confidence: 99%

“…We therefore conducted a literature review with Medline, Embase, and Cochrane library databases using the search terms “parotid,” “Küttner tumour,” and/or “chronic sclerosing sialadenitis.” Only full texts were considered for review. We identified 8 case reports of Küttner tumor affecting the parotid gland 2,5 –9 and 1 case series of 7 patients involving 1 patient with bilateral parotid involvement 9 and 1 case series of 4, one of which had parotid gland involvement. 10…”

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confidence: 99%